Treatment of Central Giant Cell Granuloma With Denosumab Therapy in Two Patients

Malmquist, Michael P.; Schow, Sterling R.

doi:10.1016/j.joms.2013.06.137

Cited by 3 publications

(4 citation statements)

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“…However, it has also been used in the treatment of other skeletal diseases such as CGCG, osteogenesis imperfecta, 25 aneurysmal bone cysts, 26,27 and fibrous dysplasia 28 . There are 22 reported patients who received denosumab for treatment of CGCG in the literature; (Table S1) 1,6,29–35 . Sixteen of the 22 patients had complete response to therapy without need for subsequent surgical intervention.…”

Section: Discussionmentioning

confidence: 99%

“…28 There are 22 reported patients who received denosumab for treatment of CGCG in the literature; (Table S1). 1,6,[29][30][31][32][33][34][35] Sixteen of the 22 patients had complete response to therapy without need for subsequent surgical intervention. One patient had partial response with stable disease that was surgically treated after 12 months of denosumab therapy.…”

Section: Discussionmentioning

confidence: 99%

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Treatment of central giant cell granuloma in children with denosumab

Choe

Smith

Okcu

et al. 2020

Pediatric Blood & Cancer

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Central giant cell granuloma (CGCG) is a benign but locally aggressive intraosseous lesion of the mandible. Historically, it is treated by curettage or resection. Medical therapy is indicated when surgery is associated with increased morbidity or in adjuvant setting to decrease recurrence. Treatment of CGCG with denosumab, a receptor activator of nuclear factor kappa‐beta (RANK) ligand inhibitor, is not well studied, especially in children. Here, we describe our experience with the use of denosumab in the treatment of six children with CGCG. All patients had a favorable response with manageable side effects, which suggests that denosumab is an effective treatment option without increased morbidity.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Treatment of central giant cell granuloma in children with denosumab

Choe

Smith

Okcu

et al. 2020

Pediatric Blood & Cancer

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“…In 2013, it was approved by the U.S. Food and Drug Administration (FDA) for treatment of GCT, (49)(50)(51) and has been given "off-label" to children with various skeletal disorders including osteogenesis imperfecta, (52) fibrous dysplasia, (53) JPD, (54) and posttransplantation hypercalcemia in OPT, (55) and to adults with GCG. (56) There would be concern for abrupt hypocalcemia, because significant hypocalcemia has complicated treatment of JPD with denosumab (54) or zolendronate, (57) and a rapid increase in his already elevated bone mass. We do not know if our patient's rapid remodeling skeleton and GCG represented an "OC-autonomous" disease treatable by marrow cell transplantation, so antiresorptive therapy clearly seemed safest.…”

Section: Discussionmentioning

confidence: 99%

“…Denosumab, an anti‐RANKL monoclonal antibody, was not available when we treated our patient with PMD. In 2013, it was approved by the U.S. Food and Drug Administration (FDA) for treatment of GCT, and has been given “off‐label” to children with various skeletal disorders including osteogenesis imperfecta, fibrous dysplasia, JPD, and posttransplantation hypercalcemia in OPT, and to adults with GCG . There would be concern for abrupt hypocalcemia, because significant hypocalcemia has complicated treatment of JPD with denosumab or zolendronate, and a rapid increase in his already elevated bone mass.…”

Section: Discussionmentioning

confidence: 99%

Rapid Skeletal Turnover in a Radiographic Mimic of Osteopetrosis

Whyte

Madson²,

Mumm

et al. 2014

Journal of Bone and Mineral Research

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Among the high bone mass disorders, the osteopetroses reflect osteoclast failure that prevents skeletal resorption and turnover leading to reduced bone growth and modeling and characteristic histopathological and radiographic findings. We report an 11-year-old boy with a new syndrome that radiographically mimics osteopetrosis but features rapid skeletal turnover. He presented at age 21 months with a parasellar, osteoclast-rich giant cell granuloma. Radiographs showed a dense skull, generalized osteosclerosis, and cortical thickening, medullary cavity narrowing, and diminished modeling of tubular bones. His serum alkaline phosphatase was > 5,000 IU/L (normal < 850). After partial resection, the granuloma re-grew but then regressed and stabilized during three years of uncomplicated pamidronate treatment. His hyperphosphatasemia transiently diminished but all bone turnover markers, especially those of apposition, remained elevated. Two years after pamidronate therapy stopped, BMD z-scores reached + 9.1 and + 5.8 in the lumbar spine and hip, respectively, and iliac crest histopathology confirmed rapid bone remodeling. Serum multiplex biomarker profiling was striking for low sclerostin. Mutation analysis was negative for activation of LRP4, LRP5, or TGFβ1 and for defective SOST, OPG, RANKL, RANK, SQSTM1, or sFRP1. Microarray showed no notable copy number variation. Studies of his non-consanguineous parents were unremarkable. The etiology and pathogenesis of this unique syndrome are unknown.

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